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Article

Tyrosyl-tRNA synthetase has a noncanonical function in actin bundling.

Nature communications - 8 Mar 2023

Ermanoska Biljana, Asselbergh Bob, Morant Laura, Petrovic-Erfurth Maria-Luise, Hosseinibarkooie Seyyedmohsen, Leitão-Gonçalves Ricardo, Almeida-Souza Leonardo, Bervoets Sven, Sun Litao, Lee LaTasha, Atkinson Derek, Khanghahi Akram, Tournev Ivaylo, Callaerts Patrick, Verstreken Patrik, Yang Xiang-Lei, Wirth Brunhilde, Rodal Avital A, Timmerman Vincent, Goode Bruce L, Godenschwege Tanja A, Jordanova Albena

Abstract excerpt

Dominant mutations in tyrosyl-tRNA synthetase (YARS1) and six other tRNA ligases cause Charcot-Marie-Tooth peripheral neuropathy (CMT). Loss of aminoacylation is not required for their pathogenicity, suggesting a gain-of-function disease mechanism. By an unbiased genetic screen in Drosophila, we link YARS1 dysfunction to actin cytoskeleton organization. Biochemical studies uncover yet unknown actin-bundling...

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Identifiers

PMID: 36890170
DOI: 10.1038/s41467-023-35908-3
PMCID: PMC9995517

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